1. A European Survey on Digestive Perianastomotic Ulcerations, a Rare Crohn-like Disorder Occurring in Children and Young Adults
- Author
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Christos Tzivinikos, Ibrahim Shamasneh, Marina Aloi, Patrizia Alvisi, Erasmo Miele, Rémi Duclaux-Loras, Jérôme Viala, Stéphanie Willot, Rosa Lima, Claire Dupont-Lucas, Mario Mašić, Julie Lemale, Daniela Prlenda-Touilleux, J. Languepin, Sanja Kolaček, Chrystèle Madre, Alexandre Fabre, Kaija-Leena Kolho, Charlotte Bergoin, Sibylle Koletzko, Jean-Pierre Hugot, Raphaël Enaud, Christine Martinez-Vinson, Maria Nachury, Alexis Mosca, Annecarin Brueckner, Emmanuelle Dugelay, Hôpital Robert Debré, University of Zagreb, Centre Hospitalier Universitaire de Saint-Etienne [CHU Saint-Etienne] (CHU ST-E), Dr von Hauner Children's Hospital [Munich, Germany], Ludwig-Maximilians-Universität München (LMU), Service de pédiatrie multidisciplinaire [Hôpital de la Timone Enfants - APHM], Hôpital de la Timone [CHU - APHM] (TIMONE), Marseille medical genetics - Centre de génétique médicale de Marseille (MMG), Aix Marseille Université (AMU)-Institut National de la Santé et de la Recherche Médicale (INSERM), Centre de recherche sur l'Inflammation (CRI (UMR_S_1149 / ERL_8252 / U1149)), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université Paris Cité (UPCité), Centro Hospitalar do Porto, CHU Bordeaux [Bordeaux], Hôpital Trousseau, Centre Hospitalier Régional Universitaire de Tours (CHRU Tours), Helsingin yliopisto = Helsingfors universitet = University of Helsinki, Geriatrics Unit [Pierre-Bénite], Université de Lyon-Centre Hospitalier Lyon Sud [CHU - HCL] (CHLS), Hospices Civils de Lyon (HCL)-Hospices Civils de Lyon (HCL), Ospedale Maggiore Carlo Alberto Pizzardi di Bologna, Nutrition, Inflammation et axe Microbiote-Intestin-Cerveau (ADEN), Université de Rouen Normandie (UNIROUEN), Normandie Université (NU)-Normandie Université (NU)-Institute for Research and Innovation in Biomedicine (IRIB), Normandie Université (NU)-Normandie Université (NU)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université de Rouen Normandie (UNIROUEN), Normandie Université (NU)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Institut National de la Santé et de la Recherche Médicale (INSERM), University of Naples Federico II = Università degli studi di Napoli Federico II, Hospices Civils de Lyon (HCL), Institute for Translational Research in Inflammation - U 1286 (INFINITE (Ex-Liric)), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), CHU Limoges, Service de Neuropédiatrie et Handicaps, Hôpital Gatien de Clocheville, CHU Tours, Université de Caen Normandie (UNICAEN), Normandie Université (NU), Institute for Research and Innovation in Biomedicine (IRIB), Normandie Université (NU)-Normandie Université (NU)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Service de Pédiatrie Médicale [Caen], Normandie Université (NU)-Normandie Université (NU)-CHU Caen, Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN)-Tumorothèque de Caen Basse-Normandie (TCBN), Al Jalila Children's Specialty Hospital, Medical Genetics Unit, Shaare Zedek Medical Center, Hevrew University Medical School, Clinicum, Children's Hospital, HUS Children and Adolescents, Madre, Chrystele, Mašić, Mario, Prlenda-Touilleux, Daniela, Brueckner, Annecarin, Koletzko, Sibylle, Fabre, Alexandre, Viala, Jérome, Lima, Rosa, Enaud, Raphael, Lemale, Julie, Kolho, Kaija-Leena, Bergoin, Charlotte, Martinez-Vinson, Christine, Dugelay, Emmanuelle, Alvisi, Patrizia, Aloi, Marina, Miele, Erasmo, Duclaux-Loras, Remi, Nachury, Maria, Languepin, Jane, Willot, Stephanie, Dupont-Lucas, Claire, Mosca, Alexi, Tzivinikos, Christo, Shamasneh, Ibrahim, Kolaček, Sanja, and Hugot, Jean-Pierre
- Subjects
Male ,intestinal resection ,Abdominal pain ,Pediatrics ,Hirschsprung disease ,Inflammatory bowel disease ,0302 clinical medicine ,3123 Gynaecology and paediatrics ,Interquartile range ,030212 general & internal medicine ,Child ,Digestive System Surgical Procedures ,Anastomosis, Surgical ,Gastroenterology ,Crohn disease ,COMPLICATION ,Short bowel syndrome ,3. Good health ,Failure to thrive ,Necrotizing enterocolitis ,Female ,digestive perianastomotic ulcerations ,030211 gastroenterology & hepatology ,gut inflammation ,medicine.symptom ,ileocaecal valve ,medicine.medical_specialty ,RESECTION ,short bowel syndrome ,Anastomosis ,Young Adult ,03 medical and health sciences ,Bloating ,medicine ,Humans ,enteral nutrition ,ANEMIA ,Ulcer ,necrotizing enterocolitis ,business.industry ,Infant, Newborn ,Infant ,ANASTOMOTIC ULCERS ,abdominal surgery ,medicine.disease ,3121 General medicine, internal medicine and other clinical medicine ,Pediatrics, Perinatology and Child Health ,business ,[SDV.MHEP]Life Sciences [q-bio]/Human health and pathology ,INFLAMMATORY-BOWEL-DISEASE - Abstract
International audience; Objectives: Digestive perianastomotic ulcerations (DPAU) resembling Crohn disease lesions are long-term complications of intestinal resections, occurring in children and young adults. They are known to be uncommon, severe and difficult to treat.Methods: In the absence of recommendations, we performed a large European survey among the members of the ESPGHAN working group on inflammatory bowel disease (IBD) in order to collect the experience of expert pediatric gastroenterologists on DPAU.Results: Fifty-one patients (29 boys and 22 girls) were identified from 19 centers in 8 countries. Most patients were followed after necrotizing enterocolitis (n = 20) or Hirschsprung disease (n = 11). The anastomosis was performed at a median age (interquartile range) of 6 [1–23] months, and first symptoms occurred 39 [22–106] months after surgery. Anemia was the most prevalent symptom followed by diarrhea, abdominal pain, bloating, and failure to thrive. Hypoalbuminemia, elevated CRP, and fecal calprotectin were common. Deep ulcerations were found in 59% of patients usually proximally to the anastomosis (68%). During a median follow-up of 40 [19–67] months, treatments reported to be the most effective included exclusive enteral nutrition (31/35, 88%), redo anastomosis (18/22, 82%), and alternate antibiotic treatment (37/64, 58%).Conclusions: Unfortunately, persistence of symptoms, failure to thrive, and abnormal laboratory tests at last follow-up in most of patients show the burden of DPAU lacking optimal therapy and incomplete understanding of the pathophysiology.
- Published
- 2021